The impact of universal newborn hearing screeningon long-term literacy outcomes: a prospectivecohort studyHannah Pimperton,1 Hazel Blythe,2 Jana Kreppner,2 Merle Mahon,3 Janet L Peacock,4

Jim Stevenson,2 Emmanouela Terlektsi,1 Sarah Worsfold,1 Ho Ming Yuen,1

Colin R Kennedy1,5

▸ Additional material ispublished online only. To viewplease visit the journal online(http://dx.doi.org/10.1136/archdischild-2014-307516).1Faculty of Medicine, Universityof Southampton, Southampton,UK2Faculty of Social and HumanSciences, University ofSouthampton, Southampton,UK3Developmental ScienceResearch Department, UCL,London, UK4Division of Health and SocialCare Research, King’s CollegeLondon, NIHR BiomedicalResearch Centre at Guy’s andSt Thomas’ NHS FoundationTrust and King’s CollegeLondon, London, UK5University of Southamptonand University HospitalSouthampton NHS FoundationTrust, Southampton, UK

Correspondence toDr Colin Kennedy, Mailpoint803G, Southampton GeneralHospital, SouthamptonSO16 6YD, UK;crk1@soton.ac.uk

Received 11 September 2014Revised 6 November 2014Accepted 9 November 2014

To cite: Pimperton H,Blythe H, Kreppner J, et al.Arch Dis Child PublishedOnline First: [please includeDay Month Year]doi:10.1136/archdischild-2014-307516

ABSTRACTObjective To determine whether the benefits ofuniversal newborn hearing screening (UNHS) seen at age8 years persist through the second decade.Design Prospective cohort study of a population sampleof children with permanent childhood hearing impairment(PCHI) followed up for 17 years since birth in periods with(or without) UNHS.Setting Birth cohort of 100 000 in southern England.Participants 114 teenagers aged 13–19 years, 76 withPCHI and 38 with normal hearing. All had previously theirreading assessed aged 6–10 years.Interventions Birth in periods with and without UNHS;confirmation of PCHI before and after age 9 months.Main outcome measure Reading comprehensionability. Regression modelling took account of severity ofhearing loss, non-verbal ability, maternal education andmain language.Results Confirmation of PCHI by age 9 months wasassociated with significantly higher mean z-scores forreading comprehension (adjusted mean difference 1.17,95% CI 0.36 to 1.97) although birth during periods withUNHS was not (adjusted mean difference 0.15, 95% CI−0.75 to 1.06). The gap between the readingcomprehension z-scores of teenagers with early comparedwith late confirmed PCHI had widened at an adjustedmean rate of 0.06 per year (95% CI −0.02 to 0.13)during the 9.2-year mean interval since the previousassessment.Conclusions The benefit to reading comprehension ofconfirmation of PCHI by age 9 months increases duringthe teenage years. This strengthens the case for UNHSprogrammes that lead to early confirmation of permanenthearing loss.Trial registration number ISRCTN03307358.

INTRODUCTIONBilateral permanent childhood hearing impairment(PCHI) of moderate, severe or profound severity isthe commonest sensory disability affecting 1 in 750children and is present at birth in more than 80%of affected children.1 PCHI of this degree has adetrimental impact on all aspects of oral languagedevelopment2–5 and impacts significantly on skillsthat depend on language ability, such as readingand writing.6 7

Identification of PCHI in early childhood enablesaffected children to receive early intervention to opti-mise their language access during a ‘sensitive period’for language development.8 More than half of babies

born with PCHI do not have prospectively identifi-able risk factors so that only universal newbornhearing screening (UNHS) programmes can identifythe majority of those affected. UNHS, when firstintroduced in the UK, more than doubled the pro-portion confirmed by 9 months to three-quarters ofall cases of bilateral PCHI >40 dB.9 10 We have pre-viously reported that children with PCHI from thatbirth cohort had significant benefits to language andreading at age 6–10 years associated with birth inperiods with UNHS and with confirmation of PCHIby age 9 months.11–13

Systematic reviews have been increasingly sup-portive of UNHS14–16 and both the UK NationalScreening Committee and the US PreventativeServices Task Force have recommended in favour ofit.17–19 During the calendar year of 2009, an esti-mated 5073 cases of PCHI were detected byUNHS in the USA, accounting for 43% of alldetected cases of the 29 medical conditions forwhich newborn screening is recommended.20

Both the US Preventative Services Task Force15 17

and a 2009 WHO report on UNHS21 have,

What is already known on this topic

▸ Universal newborn hearing screening (UNHS) isan effective way of increasing rates of earlyidentification of congenital permanentchildhood hearing impairment (PCHI).

▸ UNHS and early identification of PCHI areassociated with benefits to language andreading outcomes in middle childhood.

What this study adds

▸ This study is the first to describe the effects ofUNHS and early confirmation of PCHI onlonger-term literacy outcomes.

▸ Early confirmation of PCHI was associated withsignificant benefits to reading comprehensionin the teenage years.

▸ The benefit of early confirmation of PCHI toreading comprehension had increased frommoderate to large between the ages of 8 and17 years.

Pimperton H, et al. Arch Dis Child 2014;0:1–7. doi:10.1136/archdischild-2014-307516 1

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however, drawn attention to the evidence gap regarding benefitsbeyond primary school age and benefits to functional outcomes.This study consequently aimed to provide novel evidenceregarding the effects of UNHS and early confirmation of PCHIon functional outcomes in the teenage years. We report findingsregarding the abilities of teenagers with PCHI at age 17 yearswhom we previously assessed at age 8 years.11 12 Reading is askill that is dependent on underlying language ability22 23 thatrelates very closely to educational and employment outcomes,and as such is a key functional outcome.24 Reading comprehen-sion was therefore prespecified as the primary outcome in thisstudy.

PATIENTS AND METHODSThe children in this prospective follow-up study, 120 childrenwith bilateral PCHI >40 decibels hearing level (dB HL) (notknown to be postnatally acquired) and a comparison group of

63 normally hearing children, were drawn from a birthcohort of 157 000 children born in eight districts of southernEngland (see online supplementary appendix 1), of whomabout half were born in periods with UNHS. We previouslyreported a number of details relating to this population ininfancy and first decade, including the UNHS programmes forPCHI to which they were exposed; the service provision bydistrict and regional audiology and by other services for con-firmation and management of their PCHI; and the languageand reading abilities of the children at 6–10 years.9–13 25–31

Nine years after their previous language and reading assess-ments at 6–10 years, 76 (63%) teenagers with PCHI and 38(60%) of the normally hearing comparison group have nowparticipated in the study we report here (figure 1). We esti-mate that 49% of all oral language users with PCHI from thebirth cohort had their reading assessed at age 17.1 years (seeonline supplementary appendix 1).

Figure 1 Numbers of teenagers with permanent childhood hearing impairment who were eligible for the study and assessed for reading ability atprimary school and teenage. Greyed out section of the figure indicates the previous study at age 6–10 years. dB HL, decibels hearing level.

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ProcedureEach participant was assessed by a trained researcher, unawareof their audiological history, using the York Assessment ofReading for Comprehension Secondary Edition,32 a standar-dised reading test that provides measures of accuracy, compre-hension and summarisation skill (see online supplementaryappendix 1). A 20 min timed version33 of Raven’s StandardProgressive Matrices Plus34 was used as a measure of non-verbalability. The preplanned primary outcome of our study wasreading comprehension score after adjustment in a multiplelinear regression for severity of hearing loss, non-verbal abilityand maternal education, which were recognised as potentialconfounders of the primary outcome.11 Adjusted reading accur-acy and reading summarisation ability z-scores were preplannedsecondary outcomes.

Severity of hearing impairment was categorised from the mostrecent audiological evaluation at audiology and cochlear implantclinics as moderate (40–69 dB HL), severe (70–94 dB HL) orprofound (≥95 dB HL) according to four-frequency averaging ofthe pure-tone thresholds at 0.5, 1, 2 and 4 kHz. Maternal educa-tion was classified according to the 2001 census in the UK.

This study was approved by the Southampton and SWHampshire Research Ethics Committee. Written informedconsent for participation in the study was obtained from princi-pal caregivers and from the teenage participants themselves.

Analysis strategiesThe primary outcome (reading comprehension) and the analysisstrategy were prespecified and the statistical analysis plan waswritten before examination of the data. The target sample sizeof 96 with half of the sample born in periods with (or without)UNHS, or, in a parallel set of analyses, exposed to early (orlate) confirmation of PCHI, was sufficient to have 90% powerto detect a standardised difference in the primary outcome of atleast 0.67 SDs at a 5% significance level (two sided) using a uni-factorial test. We prespecified the definition of ‘early’ confirm-ation of PCHI as confirmation by nine completed months of

age, consistent with the definition used in our previous trial ofUNHS9 and with the US Preventive Services Task Force bench-mark for diagnosing and treating infants before 10 months ofage.15 16

The group mean and SD reading scores in the normallyhearing comparison group were used to derive z-scores for theteenagers with PCHI where the mean and SD in the normallyhearing group was 0 and 1, respectively. The z-scores in the par-ticipants with PCHI were thus expressed in terms of thenumber of SDs from the mean in the normally hearing compari-son group. Analyses were run both with and without BritishSign Language users. This did not alter the pattern of resultswhich are therefore presented for the combined group of oraland signing communicators. Where statistically significant inter-group differences were found, subgroup analysis was thenundertaken in those who had and had not received cochlearimplants. The method of adjusting reading z-scores appropri-ately to look at change in reading ability over time comparingcurrent scores with those previously obtained at aged 6–10years is described in online supplementary appendix 1.

We assessed in a linear regression model the relationshipsbetween birth during periods of UNHS or confirmation ofPCHI by age 9 months and age-adjusted reading z-scores (usingStata/SE V.12.1) in oral and signing communicators (see onlinesupplementary appendix 1). The extent to which the effect ofearly confirmation made a significant additional contribution tomodel fit after screening was included in the model was testedwith a likelihood ratio test. Normality and homogeneity of theresidual variance were examined for all measures to ensure thatthe regression models were appropriate.

RESULTSThe 114 participating teenagers were similar to the 183 whohad previously participated in the study of reading and languageat 7.9 years with regard to sex, non-verbal ability and maternaleducational level at the time of the previous study (table 1). The76 participants with PCHI (figure 1) were similar to the 120

Table 1 Demographic characteristics of participants and non-participants in the current study of reading ability in teenagers

Children with bilateral PCHI Normally hearing children

CharacteristicWhole sample*(n=120)

Teenage sample participatingin present study (n=76)

Whole sample*(n=63)

Teenage sample participatingin present study (n=38)

Mean age (SD) (range) in yearsAt primary school assessment

7.9 (1.3)(5.4 to 11.7)

7.9 (1.1)(5.8 to 10.7)

8.1 (1.0)(6.2 to 9.8)

8.0 (1.1)(6.2 to 9.8)

Female sex n (%) 53 (44) 37 (49) 26 (41) 13 (34)Severity of hearing loss n (%)

Moderate 62 (52) 38 (50) NA NASevere 29 (24) 16 (21)Profound 29 (24) 22 (29)

Born in periods with UNHS n (%) 61 (51) 37 (49) NA NAPCHI confirmed ≤9 months n (%) 57 (48) 35 (46) NA NAEnglish as main language at home n (%) 99 (83) 67 (88) 60 (95) 36 (95)Maternal education n (%)

No qualifications or <5 O-levels† 43 (36) 24 (32) 25 (40) 11 (29)≥5 O-levels or some A-levels† 62 (52) 40 (53) 25 (40) 16 (42)University or higher degree 14 (12) 12 (16) 13 (21) 11 (29)

*The ‘whole sample’ was a population-based sample of children with PCHI and a normally hearing comparison group that participated 9 years earlier in a study of language andreading at primary school age.†O-level examinations (now replaced by general certificates of education) are usually taken at 16 years of age; A-level examinations (now replaced by A2s) are taken 2 years later asqualifications for entry to higher education.NA, not applicable; PCHI, permanent childhood hearing impairment (see Patients and methods section for detailed definition of degree of PCHI); UNHS, universal newborn hearingscreening.

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who had previously participated with regard to severity ofPCHI, exposure to UNHS and confirmation of PCHI prior tonine completed months from birth (table 1). These character-istics were also similar between those who had their PCHI con-firmed by age 9 months (n=35) and those who had it confirmedlater (n=41) (table 2) and between those who were born inperiods with UNHS (n=37) and those who were not (n=39)(data not shown). The early and late confirmed PCHI groupswere similar with respect to the percentages affected by cerebralpalsy, visual disability or learning disability (table 2). Thesegroups were also similar in that the aetiology of deafness wasidentified in about half and was genetic in 34%–43% (table 2).The proportion of teenagers in whom English was not the mainlanguage at home was higher in the late (12%) than the early(3%) confirmed group and adjustment for this variable wastherefore included in the regression model in addition to thethree prespecified variables (see Patients and methods section).

The early and late confirmed groups showed mean readingcomprehension z-scores that were 0.63 and 1.74 SDs, respect-ively, below the mean reading z-score in the normally hearing

comparison group (table 3). The teenagers who had theirhearing impairment confirmed by nine completed months ofage had significantly higher adjusted mean z-scores than thelater confirmed teenagers for both reading comprehension (1.17SD) and reading summarisation (0.96 SD) (table 3). These effectsizes were larger in the 78% (51/65) who had not receivedcochlear implants (adjusted inter-group differences 1.29, 95%CI 0.52 to 2.07, p=0.002; 1.00, 95% CI 0.30 to 1.70,p=0.006, respectively). Adjusted inter-group z-score differenceson the three reading outcome measures between all teenage par-ticipants who were or were not born in periods with UNHS atbirth were smaller (0.09 to 0.22) and not statistically significant(table 3).

Change in the estimates of effect sizes and p values of earlyconfirmation and of screening was minimal when they weremodelled together rather than separately, suggesting that theseeffects were working independently (see online supplementarytable e1). Adding the effect of early confirmation into theregression model after screening was included made a significantadditional contribution to model fit (likelihood ratio test

Table 2 Characteristics of participating teenagers with hearing impairment and with normal hearing

Childrenwith bilateral PCHI (n=76)

Normally hearingchildren (n=38)Characteristic

Confirmation of PCHIat ≤9 months (n=35)

Confirmation of PCHIat >9 months (n=41)

Mean (SD) age at assessment in years 16.8 (1.5) 17.3 (1.3) 16.3 (1.2)Female sex n (%) 16 (46) 21 (51) 13 (34)Born in period with UNHS n (%) 23 (66) 14 (34) NASeverity n (%)

Moderate* 16 (45) 17 (41)Severe 7 (20) 12 (29) NAProfound 12 (34) 12 (29)

Hearing device used n (%)Cochlear implant/s 7 (20) 8 (19)Hearing aid/s 23 (66) 32 (78) NANo hearing device 5 (14)† 1 (2)‡

Mean (SD) non-verbal ability z-score§ −0.3 (0.9) −0.3 (0.8) 0 (1)

Aetiology n (%)Syndromic 9 (26) 4 (10)Other hereditary 6 (17) 10 (24) NAKnown non-genetic risk¶ 2 (6) 3 (7)Not known 18 (51) 24 (59)

Other disabilities n (%)Cerebral palsy 1 (3) 1 (2) 0Visual disability 1 (3) 1 (2) 0Learning disability 6 (17) 8 (20) 0None of the above 28 (80) 33 (80) 38 (100)

English as main language at home n (%) 34 (97) 36 (88) 36 (95)Maternal education n (%)

No qualifications/<5 O-levels** 9 (26) 10 (24) 6 (16)≥5 O-levels or some A-levels** 17 (49) 21 (51) 14 (37)University or higher degree 9 (26) 10 (24) 18 (47)

*Six participants (two with confirmation of PCHI at ≤9 months, four with confirmation of PCHI >9 months) classified with PCHI of moderate severity when previously assessed at 6–10years of age had shown improvements by the current study such that their better ear hearing thresholds now fell between 30 and 40 dB.†Three with significant additional impairments (all had chromosomal disorders and learning disability), two with moderate PCHI who were not current hearing aid users.‡One with significant additional impairments (learning disability).§Age-adjusted z-scores are listed for Ravens Progressive Matrices total score. The z-scores are the number of SDs of the scores in normally hearing children by which the age-adjustedscore differed from the mean score in the normally hearing children.¶Prematurity or cerebral palsy.**O-level examinations (now replaced by general certificates of education) are usually taken at 16 years of age; A-level examinations (now replaced by A2s) are taken 2 years later asqualifications for entry to higher education.NA, not applicable; PCHI, permanent childhood hearing impairment; UNHS, universal newborn hearing screening

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χ2=7.61, p=0.006) indicating that early confirmation of PCHIaccounted for significant unique variance in reading outcomesbeyond that accounted for by exposure to UNHS.

Comparison of the recalculated reading comprehensionz-scores at primary school age (see Patients and methods) withthose at age 13–19 years from the present study showed thatunadjusted reading comprehension z-scores remained nearlyunchanged in the early confirmed group but decreased (ie,became more negative relative to the hearing control group) inthe late confirmed group (figure 2). Compared with that in thelate confirmed group, the adjusted mean annual rate of changein the reading comprehension z-score during the 9.2 year inter-val between primary school and teenage assessments was lessnegative in the early confirmed group (mean inter-group ratedifference 0.06 per year, 95% CI −0.02 to 0.13, p=0.14). Thisinter-group difference in annual rate of change of reading com-prehension was larger and statistically significant in those whohad not received cochlear implants (mean inter-group difference0.08 per year, 95% CI 0.01 to 0.15, p=0.03).

DISCUSSIONThis study of teenagers with PCHI who were involved in trialsof UNHS is the first to describe the effects of UNHS and earlyconfirmation of PCHI on outcomes beyond the primary schoolyears. Confirmation of PCHI by nine completed months of agewas associated with significantly better performance on readingcomprehension, the prespecified primary outcome variable.Comparing the scores from the current study with readingscores obtained 9 years earlier from the same individuals, theteenagers whose PCHI had been confirmed early had main-tained their level of performance relative to the hearing controlswhereas the late confirmed teenagers had not: the gap betweenthe early and late confirmed groups had doubled between thetwo assessments. The early superiority in the reading skills ofthe early confirmed participants may have enabled them to readmore demanding reading material more frequently than theirpeers with later confirmed PCHI, thus increasing the skill gapbetween the two groups. The superiority resulting from thisrich-get-richer ‘Matthew effect’35 was more than 1 SD of therange of reading comprehension scores in their normally

Table3

Readingz-scores

forchildrenwith

bilateralPCH

Ibyageof

confirm

ationof

PCHI

andby

birth

inperiods

with

andwithoutUN

HS

Measure

Num

berof

observations

Meanz-score(SD)

Una

djustedmean

differen

ce(95%

CI)

a–b

pVa

lue

Adjusted*

mean

differen

ce(95%

CI)

a–b

pVa

lue

PCHIcon

firmed

at≤9mon

ths

(n1)

PCHIcon

firmed

at>9

mon

ths

(n2)

PCHIcon

firmed

at≤9mon

ths

(a)

PCHIcon

firmed

at>9

mon

ths(b)

YARC

readingcomprehensio

n28

37−0.63

(1.63)

−1.74

(1.50)

1.11

(0.33to

1.89)

0.006

1.17

(0.36to

1.97)

0.005

YARC

readingsummarisa

tion

2837

−0.56

(1.37)

−1.36

(1.44)

0.80

(0.09to

1.51)

0.03

0.96

(0.24to

1.68)

0.01

YARC

readingaccuracy

2733

−1.12

(1.69)

−1.71

(1.44)

0.59

(−0.22

to1.40)

0.15

0.68

(−0.09

to1.46)

0.08

UNHS

(n1)

NoUNHS

(n2)

UNHS

(a)

NoUNHS

(b)

a—b

a—b

YARC

readingcomprehensio

n33

32−1.15

(1.87)

−1.37

(1.39)

0.22

(−0.60

to1.04)

0.60

0.15

(−0.75

to1.06)

0.73

YARC

readingsummarisa

tion

3332

−0.97

(1.51)

−1.07

(1.43)

0.10

(−0.62

to0.83)

0.78

0.22

(−0.58

to1.03)

0.58

YARC

readingaccuracy

3129

−1.43

(1.59)

−1.47

(1.58)

0.04

(−0.78

to0.86)

0.93

0.09

(−0.76

to0.93)

0.84

*Adjustedforseverityof

PCHI,m

aternaleducationlevel,non-verbalabilityandEnglish

asmainlanguage

athome.

PCHI,p

ermanentchildhood

hearingimpairm

ent;UN

HS,u

niversalnewborn

hearingscreening;

YARC

,YorkAssessmentof

ReadingforComprehensio

n.

Figure 2 Unadjusted mean reading comprehension z-scores inchildren with permanent childhood hearing impairment at age 6–10 years and 13–19 years by age of confirmation of hearingimpairment. Error bars represent the 95% CI of the means in the 26early confirmed and 36 late confirmed participants who provided dataat both primary school age and teenage.

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hearing peers and is likely to impact on their life chancesthrough educational achievement and employment.24

Non-verbal ability was very similar in the early and late con-firmed groups and adjustment for it was included in the regres-sion model. This suggests that the deficit in reading scores in thelate confirmed participants did not result from a general cogni-tive deficit but rather from the specific impact of delayed accessto optimal language input early in life on language-related abil-ities. The early and late confirmed groups did not show differ-ent proportions of genetic and non-genetic aetiologies ofdeafness nor of disabilities additional to deafness that mightaccount for the observed differences in reading z-scores.

Factors other than age at confirmation of PCHI appeared todetermine reading outcomes for that minority of participantswho had received cochlear implants36 although this subgroupanalysis was not preplanned and should be treated with caution.A greater dependency of teenage reading ability of theimplanted subgroup on age at implantation than on age at con-firmation may explain this difference but studies of largernumbers of cochlear implantees are needed to determine this.

The effects of early confirmation were seen in those born inperiods with and without UNHS and the effect of UNHSappears to be dependent on the increase in rates of early con-firmation of PCHI to which it leads. The same NHS district andregional audiology teams delivered, in almost all cases, the careof both screened and unscreened and of both early and late con-firmed populations in this study31 and the different outcomesbetween these groups are likely to reflect the effect of UNHSand of early confirmation rather than any differences in the ser-vices to which they were exposed. A 2013 birth cohort in theUK would, nevertheless, be likely to show a much stronger rela-tionship between birth in periods with UNHS and reading out-comes. Effective postscreening audiology and other services forthose screening positive for PCHI in the newborn period, whichwere largely absent in the period from 1992 to 1997 for thepopulation described in this report, are now in place18 19 andtherefore screening positive on UNHS in the UK would bemore likely to lead to confirmation of PCHI by age 9 months.

The annual attrition rate (ie, 3% over 17 years since UNHSor 4% over the 9 years since assessment at primary school)among children with PCHI eligible for the present study is lowfor a teenage population with a chronic medical condition butlimited the power of the study to examine change in readingcomprehension between the primary school and teenage assess-ments. In spite of this limitation in power, the inter-group dif-ferences on the prespecified primary outcome of readingcomprehension were large enough to be both statistically signifi-cant and clinically important.

CONCLUSIONSAs the Millenium Development Goals project approaches its2015 target, UNESCO, UNICEF, the World Bank and WHO areincreasingly considering early child development, in whichinfant hearing is a critical component, as a key determinant ofsubsequent health21 37 and this report is therefore timely.Confirmation of PCHI by nine completed months of age wasassociated with significantly better performance on reading com-prehension, the prespecified primary outcome variable, and theeffect size of this benefit of early confirmation of PCHI hadincreased from moderate to large between assessments at theages of 8 and 17 years. This strengthens the case for nationalgovernments to fund UNHS programmes that increase the ratesof early confirmation of PCHI in the many developed and

developing countries where UNHS for PCHI is currently underdiscussion but not yet adopted as national policy.38–40

Acknowledgements We thank the research assistants Eleanore Coulthard, JoannePickersgill, Lisa Shipway and Zahra Taghizadeh; the audiologists Margaret Baldwin,Alyson Bumby, Adrian Dighe, Harpreet Nijar, David Reed, Joy Roberts, SueRobinson, Salim Suleman, Rosbin Syed and Huw Thomas; and the other medicaland educational professionals who supported this study. We thank particularly theparticipating teenagers and their families.

Contributors HP oversaw the conduct and analysis of the study, drafted the initialmanuscript and approved the final manuscript. JK, MM, JS and SW assisted in thedesign and supervision of the study, assisted with manuscript preparation andapproved the final manuscript. JLP assisted in the design and supervision of thestudy, supervised the statistical analysis, assisted with manuscript preparation andapproved the final manuscript. HB and ET assisted in the supervision of the studyand approved the final manuscript. HMY undertook the statistical analysis andapproved the final manuscript. CRK designed and supervised the study, assisted inmanuscript preparation and approved the final manuscript.

Funding This work was funded by The Wellcome Trust (Grant number 089251/Z/09/Z) which had no role in the design, conduct of the study, collection analysis andinterpretation of the data, preparation, review or approval of the manuscript, or thedecision to submit the manuscript for publication.

Competing interests None.

Ethics approval Southampton and SW Hampshire Research Ethics Committee.

Provenance and peer review Not commissioned; externally peer reviewed.

Data sharing statement The authors are willing to share all unpublished datafrom the study with bona fide researchers. The database can be made available tothem through discussion with the corresponding author and the Wellcome Trust.

Open Access This is an Open Access article distributed in accordance with theterms of the Creative Commons Attribution (CC BY 4.0) license, which permitsothers to distribute, remix, adapt and build upon this work, for commercial use,provided the original work is properly cited. See: http://creativecommons.org/licenses/by/4.0/

REFERENCES1 Davis A, Bamford J, Wilson I, et al. A critical review of the role of neonatal hearing

screening in the detection of congenital hearing impairment. Health Technol Assess1997;1:i–iv, 1–176.

2 Eisenberg LS. Current state of knowledge: speech recognition and production inchildren with hearing impairment. Ear Hear 2007;28:766–72.

3 Moeller MP, Tomblin JB, Yoshinaga-Itano C, et al. Current state of knowledge:language and literacy of children with hearing impairment. Ear Hear2007;28:740–53.

4 Luckner JL, Cooke C. A summary of the vocabulary research with students who aredeaf or hard of hearing. Am Ann Deaf 2010;155:38–67.

5 Luckner JL, Handley CM. A summary of the reading comprehension researchundertaken with students who are deaf or hard of hearing. Am Ann Deaf2008;153:6–36.

6 Wauters LN, Van Bon WHJ, Tellings A. Reading comprehension of Dutch deafchildren. Read Writ 2006;19:49–76.

7 Conrad R. The deaf school child. London: Harper Row, 1979.8 Thomas MSC, Johnson MH. New advances in understanding sensitive periods in

brain development. Curr Dir Psychol 2008;17:1–5.9 Kennedy CR, Kimm L, Dees DC, et al. Controlled trial of universal neonatal

screening for early identification of permanent childhood hearing impairment.Lancet 1998;352:1957–64.

10 Kennedy CR, McCann D, Campbell MJ, et al. Universal newborn screening forpermanent childhood hearing impairment: an 8-year follow-up of a controlled trial.Lancet 2005;366:660–2.

11 Kennedy CR, McCann DC, Campbell MJ, et al. Language ability after early detectionof permanent childhood hearing impairment. NEngl J Med 2006;354:2131–41.

12 McCann DC, Worsfold S, Law CM, et al. Reading and communication skills afteruniversal newborn screening for permanent childhood hearing impairment. Arch DisChild 2009;94:293–7.

13 Worsfold S, Mahon M, Yuen HM, et al. Narrative skills following early confirmationof permanent childhood hearing impairment. Dev Med Child Neurol2010;52:922–8.

14 Pimperton H, Kennedy CR. The impact of early identification of permanentchildhood hearing impairment on speech and language outcomes. Arch Dis Child2012;97:648–53.

15 Nelson HD, Bougatsos C, Nygren P. Universal newborn hearing screening:systematic review to update the 2001 US Preventive Services Task ForceRecommendation. Pediatrics 2008;122:E266–E76.

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group.bmj.com on November 26, 2014 - Published by http://adc.bmj.com/Downloaded from

16 Thompson DC, McPhillips H, Davis RL, et al. Universal newborn hearingscreening—Summary of evidence. JAMA-J Am Med Assoc 2001;286:2000–10.

17 Calonge N, Petitti DB, DeWitt TG, et al. Universal screening for hearing loss innewborns: US preventive services task force recommendation statement. Pediatrics2008;122:143–8.

18 Bamford J, Uus KI, Davis A. Screening for hearing loss in childhood: issues,evidence and current approaches in the UK. J Med Screen 2005;12:119–24.

19 Public Health England. NHS Newborn Hearing Screening Programme 2013 [cited2013 06.09]; http://hearing.screening.nhs.uk/

20 Howell RR, Terry S, Tait VE, et al. CDC grand rounds: newborn screening andimproved outcomes. MMWR Morb Mortal Wkly Rep 2012;61:390–3.

21 World Health Organization. Newborn and infant hearing screening: Current issuesand guiding principles for action. Geneva: WHO Press, 2009.

22 Fricke S, Bowyer-Crane C, Haley AJ, et al. Efficacy of language intervention in theearly years. J Child Psychol Psychiatry 2013;54:280–90.

23 Tunmer WE, Chapman JW. The simple view of reading redux: vocabularyknowledge and the independent components hypothesis. J Learn Disabil2012;45:453–66.

24 Dugdale G, Clark C. Literacy changes lives: an advocacy resource. London: NationalLiteracy Trust; 2008.

25 Watkin PM, Baldwin M. Confirmation of deafness in infancy. Arch Dis Child1999;81:380–9.

26 Tucker SM, Bhattacharya J. Screening of hearing impairment in the newborn usingthe auditory response cradle. Arch Dis Child 1992;67:911–19.

27 Petrou S, McCann D, Law CM, et al. Health status and health-related quality of lifepreference-based outcomes of children who are aged 7 to 9 years and havebilateral permanent childhood hearing impairment. Pediatrics 2007;120:1044–52.

28 Schroeder L, Petrou S, Kennedy CR, et al. The economic costs of congenital bilateralpermanent childhood hearing impairment. Pediatrics 2006;117:1101–12.

29 Stevenson J, McCann D, Watkin P, et al. The relationship between languagedevelopment and behaviour problems in children with hearing loss. J Child PsycholPsychiatry 2010;51:77–83.

30 Stevenson J, McCann DC, Law CM, et al. The effect of early confirmation ofhearing loss on the behaviour in middle childhood of children with bilateral hearingimpairment. Dev Med Child Neurol 2011;53:269–74.

31 Watkin P, McCann D, Law C, et al. Language ability in children with permanenthearing impairment: the influence of early management and family participation.Pediatrics 2007;120:E694–701.

32 Stothard SE, Hulme C, Clarke P, et al. YARC York Assessment of Reading forComprehension Secondary. GL Assessment; 2010.

33 Hamel R, Schmittman VD. The 20-minute version as a predictor of the RavenAdvanced Progressive Matrices Test. Educ Psychol Meas 2006;66:1039–46.

34 Styles I, Raven M, Raven JC. Standard progressive matrices—plus version. London:Pearson; 1998.

35 Stanovich KE. Matthew effects in reading: some consequences of individualdifferences in the acquisition of literacy. Read Res Q 1986;21:360–407.

36 Markman TM, Quittner AL, Eisenberg LS, et al. Language development aftercochlear implantation: an epigenetic model. J Neurodev Disord 2011;3:388–404.

37 Olusanya BO. Highlights of the new WHO Report on Newborn and infant hearingscreening and implications for developing countries. Int J Pediatr Otorhinolaryngol2011;75:745–8.

38 Liang Q, Mason B. Enter the dragon—China’s journey to the hearing world.Cochlear Implants Int 2013;14(Suppl 1):S26–31.

39 Olusanya BO. Neonatal hearing screening and intervention in resource-limitedsettings: an overview. Arch Dis Child 2012;97:654–9.

40 The Canadian Association of Speech-Language Pathologists and Audiologists.Universal Newborn Hearing Screening. 2013 [cited 2013 22.12]; http://www.caslpa.ca/caslpa-work/universal-newborn-hearing-screening

Pimperton H, et al. Arch Dis Child 2014;0:1–7. doi:10.1136/archdischild-2014-307516 7

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prospective cohort studyscreening on long-term literacy outcomes: a The impact of universal newborn hearing

Ming Yuen and Colin R KennedyPeacock, Jim Stevenson, Emmanouela Terlektsi, Sarah Worsfold, Ho Hannah Pimperton, Hazel Blythe, Jana Kreppner, Merle Mahon, Janet L

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